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Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin

OBJECTIVES: Cytomegalovirus (CMV)‐related encephalitis is a rare but potentially life‐threatening complication of CMV infection in immunocompromised patients. The high mortality rate is associated with deficient immune system reconstitution after hematopoietic stem cell transplant (HSCT) and poor bi...

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Autores principales: Maximova, Natalia, Marcuzzi, Annalisa, Del Rizzo, Irene, Zanon, Davide, Maestro, Alessandra, Barbi, Egidio, Sala, Roberto
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7670254/
https://www.ncbi.nlm.nih.gov/pubmed/33235734
http://dx.doi.org/10.1002/cti2.1201
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author Maximova, Natalia
Marcuzzi, Annalisa
Del Rizzo, Irene
Zanon, Davide
Maestro, Alessandra
Barbi, Egidio
Sala, Roberto
author_facet Maximova, Natalia
Marcuzzi, Annalisa
Del Rizzo, Irene
Zanon, Davide
Maestro, Alessandra
Barbi, Egidio
Sala, Roberto
author_sort Maximova, Natalia
collection PubMed
description OBJECTIVES: Cytomegalovirus (CMV)‐related encephalitis is a rare but potentially life‐threatening complication of CMV infection in immunocompromised patients. The high mortality rate is associated with deficient immune system reconstitution after hematopoietic stem cell transplant (HSCT) and poor bioavailability of antiviral drugs in cerebrospinal fluid (CSF). CMV‐related central nervous system (CNS) infection may occur with aspecific symptoms, without evidence of either blood viral load or magnetic resonance imaging (MRI) signs of encephalitis. METHODS: Here, we describe a 10‐year‐old girl who underwent an allogeneic HSCT and subsequently developed CMV encephalitis. Because of the absence of CMV antigen in the blood, the diagnosis of encephalitis was proposed only after a delay, following the onset of immune reconstitution inflammatory syndrome (IRIS). Two months of combined dual antiviral therapy with ganciclovir and foscarnet proved ineffective against CMV and caused significant bone marrow and renal toxicity. To avoid further toxicity, the girl was given daily treatment with CMV‐hyperimmune globulins alone. RESULTS: After three weeks, the CSF viral load dropped significantly and was undetectable within three more weeks. In the meantime, the renal impairment resolved, and there was a complete bone marrow recovery. CONCLUSION: We suggest that this patient succeeded in achieving CMV CSF clearance with high dose of CMV‐hyperimmune globulin, given alone, because of the ability of immunoglobulins to penetrate the blood–brain barrier (BBB).
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spelling pubmed-76702542020-11-23 Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin Maximova, Natalia Marcuzzi, Annalisa Del Rizzo, Irene Zanon, Davide Maestro, Alessandra Barbi, Egidio Sala, Roberto Clin Transl Immunology Case Reports OBJECTIVES: Cytomegalovirus (CMV)‐related encephalitis is a rare but potentially life‐threatening complication of CMV infection in immunocompromised patients. The high mortality rate is associated with deficient immune system reconstitution after hematopoietic stem cell transplant (HSCT) and poor bioavailability of antiviral drugs in cerebrospinal fluid (CSF). CMV‐related central nervous system (CNS) infection may occur with aspecific symptoms, without evidence of either blood viral load or magnetic resonance imaging (MRI) signs of encephalitis. METHODS: Here, we describe a 10‐year‐old girl who underwent an allogeneic HSCT and subsequently developed CMV encephalitis. Because of the absence of CMV antigen in the blood, the diagnosis of encephalitis was proposed only after a delay, following the onset of immune reconstitution inflammatory syndrome (IRIS). Two months of combined dual antiviral therapy with ganciclovir and foscarnet proved ineffective against CMV and caused significant bone marrow and renal toxicity. To avoid further toxicity, the girl was given daily treatment with CMV‐hyperimmune globulins alone. RESULTS: After three weeks, the CSF viral load dropped significantly and was undetectable within three more weeks. In the meantime, the renal impairment resolved, and there was a complete bone marrow recovery. CONCLUSION: We suggest that this patient succeeded in achieving CMV CSF clearance with high dose of CMV‐hyperimmune globulin, given alone, because of the ability of immunoglobulins to penetrate the blood–brain barrier (BBB). John Wiley and Sons Inc. 2020-11-17 /pmc/articles/PMC7670254/ /pubmed/33235734 http://dx.doi.org/10.1002/cti2.1201 Text en © 2020 The Authors. Clinical & Translational Immunology published by John Wiley & Sons Australia, Ltd on behalf of Australian and New Zealand Society for Immunology, Inc. This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Reports
Maximova, Natalia
Marcuzzi, Annalisa
Del Rizzo, Irene
Zanon, Davide
Maestro, Alessandra
Barbi, Egidio
Sala, Roberto
Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title_full Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title_fullStr Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title_full_unstemmed Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title_short Standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
title_sort standard treatment–refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus‐specific hyperimmune globulin
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7670254/
https://www.ncbi.nlm.nih.gov/pubmed/33235734
http://dx.doi.org/10.1002/cti2.1201
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